High-throughput tracking enables systematic phenotyping and drug repurposing in disease models.

There are thousands of Mendelian diseases with more being discovered weekly and the majority have no approved treatments. To address this need, we require scalable approaches that are relatively inexpensive compared to traditional drug development. In the absence of a validated drug target, phenotypic screening in model organisms provides a route for identifying candidate treatments.

The zebrafish mutant implicates sodium homeostasis in sleep regulation.

Sleep is a nearly universal feature of animal behaviour, yet many of the molecular, genetic, and neuronal substrates that orchestrate sleep/wake transitions lie undiscovered. Employing a viral insertion sleep screen in larval zebrafish, we identified a novel gene, (), whose loss results in behavioural hyperactivity and reduced sleep at night. The neuronally expressed gene is conserved across vertebrates and encodes a small single-pass transmembrane protein that is structurally similar to the Na,K-ATPase regulator, FXYD1/Phospholemman.

Megapixel camera arrays enable high-resolution animal tracking in multiwell plates.

Tracking small laboratory animals such as flies, fish, and worms is used for phenotyping in neuroscience, genetics, disease modelling, and drug discovery. An imaging system with sufficient throughput and spatiotemporal resolution would be capable of imaging a large number of animals, estimating their pose, and quantifying detailed behavioural differences at a scale where hundreds of treatments could be tested simultaneously. Here we report an array of six 12-megapixel cameras that record all the wells of a 96-well plate with sufficient resolution to estimate the pose of C.

Behavioral fingerprints predict insecticide and anthelmintic mode of action.

Novel invertebrate-killing compounds are required in agriculture and medicine to overcome resistance to existing treatments. Because insecticides and anthelmintics are discovered in phenotypic screens, a crucial step in the discovery process is determining the mode of action of hits. Visible whole-organism symptoms are combined with molecular and physiological data to determine mode of action.

Zebrafish sleep: from geneZZZ to neuronZZZ.

All animals have a fundamental and unavoidable requirement for rest, yet we still do not fully understand the processes that initiate, maintain, and regulate sleep. The larval zebrafish is an optically translucent, genetically tractable model organism that exhibits sleep states regulated by conserved sleep circuits, thereby offering a unique system for investigating the genetic and neural control of sleep. Recent studies using high throughput monitoring of larval sleep/wake behaviour have unearthed novel modulators involved in regulating arousal and have provided new mechanistic insights into the role of established sleep/wake modulators.